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Frustration and Emotional Regulation in Nonsyndromic Craniosynostosis

an fMRI Study

Wu, Robin T. BSa; Yang, Jenny F. MDa,b; Zucconi, William DOc; Lacadie, Cheryl BSd; Swallow, Matthew BAa; Sun, Alexander H. BSa; Eilbott, Jeffreyd; Mayes, Linda C MDe; Steinbacher, Derek M. MD, DMD; Pelphrey, Kevin PhDd; Persing, John A. MDb

Plastic and Reconstructive Surgery: May 8, 2019 - Volume PRS Online First - Issue - p
doi: 10.1097/PRS.0000000000005850
Original Article: PDF Only

Purpose: Nonsyndromic craniosynostosis (NSC) may manifest with complex behavioral, attentional, and emotional sequelae. We characterized higher-level brain connectivity in adolescent NSC patients in response to emotional frustration.

Methods: Surgically corrected patients age >9 with NSC were age/gender/handedness matched to controls. Patients participated in a GoNoGo task, structured as "win/lose/recovery" paradigms; "Win" allowed point increases, "lose" led to net negative points, and "recovery" allowed re-accumulation. BioImage Suite was used to analyze whole-brain intrinsic connectivity between tasks with cluster-corrected group-level T-maps. P<0.05 was significant.

Results: Seven unilateral-coronal (ULC; average age 12.2 years), six metopic (average age 11.5 years), and respective matched controls were included. ULC had worse emotional regulation scores on the Behavior Rating Inventory of Executive Function survey(p=0.065) and performed poorly on the GoNoGo task(p<0.001). Metopics had four regions of interest (ROI) with majority decreased activity compared to controls, and few differences between tasks. ULC had eleven ROIs, majority decreased during "win" and "lose", but all increased during "recovery". Metopic patients had decreased blood-oxygenation-level-dependent signal in the posterior cingulate(p=0.017) and middle temporal gyrus(MTG;p=0.042). ULC had decreased signal in the posterior cingulate(p=0.023), MTG(p=0.027), and thalamus(p=0.033), but increased signal in the cuneus(p=0.009) and cerebellum(p=0.009). R-ULC, but not metopic/controls, had increased right brain activity in the caudate(p=0.030), thalamus(p=0.011), temporal lobe(p=0.012), and cerebellum(p=0.029).

Conclusions: ULC patients may have emotional dyregulation in response to frustration while metopic patients may have attenuated emotional reactions. Evidence of R-ULC brain laterality suggest that the area of suture fusion may contribute to the mechanism of dysfunction.

a Yale School of Medicine

b Department of Surgery, Section of Plastic Surgery, Yale School of Medicine

c Department of Radiology, Yale School of Medicine

d Magnetic Resonance Research Center, Yale School of Medicine

e Yale Child Study Center, Yale School of Medicine

Presented at: Plastic Surgery Research Council (PSRC) 2018 in Birmingham, Alabama

FD - No financial or commercial conflicts of interests for any other authors.

Corresponding Author: John A. Persing, MD, Department of Surgery, Section of Plastic Surgery, Yale University School of Medicine, P.O. Box 8041, New Haven, CT 06520-8062 203.785.2571,

©2019American Society of Plastic Surgeons